1

Introduction

The first documentation of ureterocele by Lechler in 1835 described it as a “double bladder” found during a postmortem of a 3-month-old infant. Subsequent reports expanded on this, with Bostroem detailing multiple cases in 1884, and terms like “inverted ureter”, “cystic dilation of the vesical end of the ureter”, and “ureter ballooning” being introduced. Stoeckel coined the term “ureterocele” in 1907, defining it as a ureteral hernia.

Ureterocele incidence ranges from 1 in 500 to 1 in 4000 autopsy cases, ^, with a higher prevalence in females (4–6 times more common) and caucasians. ^, Ureteroceles show no side preference and are bilateral in 10 % of cases. These cystic dilations of the submucosal terminal ureter can present unilaterally or bilaterally, orthotopically or ectopically, and in single or duplicated urinary systems, complicating diagnosis and management. While more frequent in females, ^, massive ureterocele in a male neonate with urinary retention is exceptionally rare, underscoring the need for timely diagnosis and surgical intervention to prevent long-term complications.

2

Case report

A 2-day-old male neonate presented with complaints of inadequate urine output and visible abdominal distension. Although abundant diuresis was noted at birth, there was a subsequent cessation of diuresis for approximately 18 hours of life, accompanied by a palpable bladder upon abdominal examination, which raised suspicion for urinary retention. Subsequent urinary catheterization followed with continuous bladder drainage using a suprapubic vesicostomy system (SVD) was performed to confirm acute urinary retention.

Antenatal ultrasonography had revealed duplication with grade IV hydronephrosis on the left side and grade II on the right side. This finding was confirmed by repeated diagnostic imaging studies within the first two days of life, which demonstrated bilateral hydronephrosis along with a sizable cystic lesion within the bladder and a left duplex collecting system ( Fig. 1 ).

Preoperative US with moderate hydronephrosis on right kidney (A), duplicity and severe hydronephrosis on left kidney (B) and bladder with ureterocele (C).

Further evaluation via voiding cystourethrogram heightened suspicion for the presence of a intravesical massive ureterocele contributing to the urinary obstruction in this male neonate ( Fig. 2 ).

VCUR evidenciating cecoureterocele.

Timely surgical intervention was initiated, commencing with cystoscopy. Evaluation of the anterior and posterior urethra revealed no lesions or areas of stenosis, ruling out posterior urethral valve as well. Upon approaching the bladder neck, a large ureterocele on the left side obstructing the internal urethral meatus was identified. Right ureteral orifice appearing normal and both left ureteral meatus were noted to be posterior to the ureterocele (Video). Subsequent “watering-can” puncture of the ureterocele was performed, resulting in successful alleviation of the obstruction and restoration of normal urine flow.

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